Literature DB >> 34131769

Hypophosphatasia and cleidocranial dysplasia-a case report and review of the literature: the role of the neurosurgeon.

Alexandros Blionas1, Gerhard M Friehs2, Vasileios A Zerris2.   

Abstract

Hypophosphatasia (HPT) and cleidocranial dysplasia (CCD) are rare genetic disorders characterized by both defective ossification and bone mineralization. Patients usually present with craniosynostosis and cranial defects which in many cases require surgical repair. There is only 1 reported case of combined HPT and CCD in the literature. Our reported case involves a 3.5-year-old girl with concomitant homozygous CCD and heterozygous HPT. The child had an extended cranial defect since birth which improved with the administration of Strensiq and was followed until preschool age. Bone defects were relatively minor on revaluation. Due to the limited final defect, we decided not to intervene. In HPT-CCD patients, bone defects are overestimated due to osteomalacia, and thus, management strategy should be less aggressive. They should undergo surgical repair with cranioplasty with the use of cement and/or titanium meshes in case of extended final defects.
© 2021. The Author(s), under exclusive licence to Springer-Verlag GmbH Germany, part of Springer Nature.

Entities:  

Keywords:  Cleidocranial; Cranial; Craniosynostosis; Defect; Genetic; Hypophosphatasia

Mesh:

Year:  2021        PMID: 34131769     DOI: 10.1007/s00381-021-05261-1

Source DB:  PubMed          Journal:  Childs Nerv Syst        ISSN: 0256-7040            Impact factor:   1.475


  3 in total

1.  Hypophosphatasia: screening and family investigation.

Authors:  I Rubecz; K Méhes; L Klujber; L Bozzay; J Weisenbach; J Fenyvesi
Journal:  Clin Genet       Date:  1974       Impact factor: 4.438

2.  Severe cleidocranial dysplasia can mimic hypophosphatasia.

Authors:  Sheila Unger; Etienne Mornet; Stefan Mundlos; Susan Blaser; David E C Cole
Journal:  Eur J Pediatr       Date:  2002-06-28       Impact factor: 3.183

3.  Cleidocranial dysplasia with decreased bone density and biochemical findings of hypophosphatasia.

Authors:  Eva Morava; Judit Kárteszi; János Weisenbach; Almuth Caliebe; Stefan Mundlos; Károly Méhes
Journal:  Eur J Pediatr       Date:  2002-10-09       Impact factor: 3.183

  3 in total

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